Int J Oral-Med Sci 10(1):31-36,2011 Original Article

Traumatic of the Diagnosed Using Contrast-enhanced Magnetic

Resonance Imaging

Machiyo Yokokawa,Shigeo Tanaka,Makiko Ono,Sayaka Sakamoto,Miya Kato, Takashi Kaneda,Tadahiko Utsunomiya,Hirotsugu Yamamoto,Masamichi Komiya, and Yoshiaki Akimoto

Departments of Oral , Radiology,and Oral Pathology,Nihon University School of Dentistry at Matsudo, Matsdo,Chiba 271-8587,Japan Department of Oral Surgery,Nihon University School of Medicine,Itabashi,Tokyo 173-8610,Japan

Correspondence to: Machiyo Yokokawa Traumatic bone cyst(TBC)is an uncommon nonepithelial-lined cavity

E-mail: yokokawa.machiyo＠nihon of the .In the clinical diagnosis of TBC, it may be difficult to

-u.ac.jp differentiate TBC from other cysts and tumors.We described a case of

TBC of the mandible with magnetic resonance imaging findings,the

radiological and clinical characteristics, and the surgical findings. Keywords: Contrast-enhanced magnetic resonance images provided useful infor- traumatic bone cyst,mandible,MRI mation for distinguishing TBC from other cysts and tumors.

In the clinical diagnosis of TBC,it may be difficult

A traumatic bone cyst (TBC) is an uncommon to differentiate TBC from cysts and tumors, espe- nonepithelial-lined cavity of the jaws(1-5).In 1929, cially when TBC appears with a unilocular shape. Lucas and Blum for the first time described TBC as Recently, magnetic resonance imaging (MRI) has a separate disease entity (6).However, it was not been used in the diagnosis and definition of cystic until 1946 that the diagnostic criteria of this cyst lesions in the oral and maxillofacial regions (17,24, were established.These criteria remain valid today 26). and comprise a generally single lesion without an Thus,we described a case of TBC of the mandible epithelial lining,surrounded by bony walls and either with MRI findings,the radiological and clinical char- lacking contents or containing liquid and/or connec- acteristics,and the surgical findings. tive tissue (7). In the classification of the World

Health Organization (WHO),TBCs are included in the group of bone-related tumor-like lesions, A 12-year-old girl was referred by a dentist to the together with aneurysmal bone cyst,juxta-articular Oral Surgery Department for evaluation of a bone cyst (intraosseous ganglion), metaphyseal unilocular radiolucency of the anterior mandible fibrous defect (nonossifying ), eosinophilic accompanied with vague pain that had begun the granuloma, fibrous dysplasia, myositis ossificans, previous week. Intraoral examination did not show and brown tumor of hyperparathyroidism (8,9). any soft tissue abnormality or bony expansion.The

Many terms have been used to describe the TBC periodontium was noted to be healthy with no evi- such as solitary bone cyst(1-3,7,10-18),simple bone dence of or mobility.There were no cyst (4,19-26),hemorrhagic bone cyst (27-31),pro- carious lesions.The anterior mandibular teeth were gressive bone cyst (32),idiopathic bone cyst (33,34), vital and they responded normally to electrical pulp and unicameral bone cyst(35-37),in which traumatic testing. bone cyst is the general term that better describes Clinical examination was unremarkable with no this cystic lesion (5,38-51). evidence of lymphadenopathy and the patient Int J Oral-Med Sci 10(1):31-36,2011 revealed no contributory medical history. On radiographic examination, a panoramic view revealed an oval unilocular radiolucency extending from the inferior aspect of the apices of the lower right canine to the left canine (Fig.1).An occlusal view showed a scalloped radiolucency between the teeth in the radiolucent area(Fig.2).Computed tomo- graphy axial scanning demonstrated the presence of a unicystic lesion at the mandibular symphysis, where the lingual cortical plate showed slight expan- Fig.1. A preoperative panoramic radiograph revealed a well sion and thinning (Fig.3). Magnetic resonance -defined unilocular radiolucency(arrows). images further confirmed the details. T1-weighted axial magnetic resonance imaging with gadolinium showed the peripheral area of the lesion rim enhance- ment and slight enhancement of the inner part of the cyst cavity (Fig.4). T2-weighted axial magnetic resonance imaging demonstrated a homogenous high signal intensity area,which indicated fluid-like sig- nal intensity(Fig.4). A clinical diagnosis of TBC was made and the operation for enucleation and curettage of the cystic lesion was performed under general anesthesia. A mucoperiosteal flap was raised,exposing the labial surface of the alveolar bone overlying the cystic area.None of the surface presented any noticeable bony expansion.A window was made with a surgical bur in order to reach the lesion.The bony cavity was filled with fluid and there was lining on its walls apart from an extremely thin layer of connecting Fig.2. An occlusal radiograph showing a unilocular tissue.Following careful curettage,small bone chips radiolucency with a thinly corticated margin(arrows). with parts of the membrane were submitted for microscopic examination. Because the operative findings were highly suggestive of the diagnosis of

TBC, no further treatment was done apart from curettage.The postoperative course was uneventful. A follow-up panoramic radiograph at 6 months has shown good bony filling of the midline region of the mandible(Fig.5). Histological examination revealed the thin cystic wall to be a connective tissue membrane with dilated blood vessels with deposits of hemosiderin granules and mucinous degeneration in the inner side and compact bone in the outer side.There was no epith- Fig.3. Axial computed tomography demonstrates a slight elial lining (Figs.6 and 7).The final diagnosis of expansion of bone on the lingual side of the mandible(arrows). Int J Oral-Med Sci 10(1):31-36,2011

Fig.4. T1-weighted axial magnetic resonance imaging with gadolinium demonstrates the peripheral area of the lesion

rim enhancement (arrow)(left).T2-weighted axial magnetic resonance imaging demonstrates a homogenous high signal

intensity area(arrow)(right).

Fig.5. Panoramic radiograph at 6 months after surgery showed good bony filling of the midline region of the mandible.

Fig.7. Dilated blood vessels and hemosiderin granules in the

fibrous connective tissues of the cyst wall(arrows,hemosider- in granules; asterisks, blood vessels) (original magnifica- tion ×600).

TBC was made from the histological examination, clinical course,and the operative findings.

Traumatic bone cysts are mainly found in young

persons most frequently during the second and third

decades of life(16,20,37-39,43,45).The reported sex

distributions vary: even(39,45),more frequently in

men (20,29, 37,38),and more frequently in women (21,47,50).The majority of TBCs are located in the Fig.6. The cyst wall consists of fibrous connective tissue and mandibular body between the canine and the third compact bone (B, bone; arrows, fibrous connective tissue) (original magnification, ×100). molar (16,20,27,29).The mandibular symphysis is Int J Oral-Med Sci 10(1):31-36,2011 the second most common site (13-14.3%) (16, 50). tion (45).Although the mean age at presentation is

Fewer cases are reported in the ramus,condyle,and the second and third decades, when it could be the maxilla (11, 12, 15, 22, 23, 29, 32, 39, 41, 49). hypothesized that trauma to the jaws is more likely, Clinically,TBC is asymptomatic in most cases and is there is no difference in the prevalence between often accidentally discovered on routine radiological males and females or the prevalence is higher in examination(29,38,47).Pain is the presenting symp- females despite a higher incidence of trauma in tom in 10% to 30% of the patients (27,29, 39, 47). males.No history of trauma could be elicited from

Other,more unusual symptoms include tooth sensi- our patient.Cohen proposed that the formation and tivity (20,39),paresthesia (40,47),fistulas (20),and existence of the traumatic bone cyst are due to a pathologic fracture of the mandible (28, 32).Often blockage of the normal draining of interstitial fluid. noted is expansion of the cortical plate of the - Because the normal hemodynamic pressures of the bone, usually buccally or labially, resulting in area are low,the expansion of the cyst would require intraoral and extraoral swelling.The teeth adjacent only a small increase in the hydrodynamic pressure to the lesion are usually vital without mobility, within the cyst (19).Unfortunately,as in our case, displacement,or resorption of their roots (20,27,29, many traumatic bone cysts are found to be empty at

39,47).On radiological examination,TBC appears as surgery with no evidence of cyst fluid. This would a unilocular radiolucency with an irregular but well seem to challenge this proposal.Furthermore,if the defined (or partly well defined) outline, with or cyst developed because of a blockage of draining without sclerotic lining around the periphery of the interstitial fluid,one might expect that these lesions lesion. Characteristic for TBC is the “scalloping would develop with a more equal frequency in all effect”extending between the roots of the teeth (16, locations within the facial skeleton rather than oc- 39).On MRI examination of TBC cases,the contrast curring with a higher frequency in the posterior

-enhanced T1WI of Gd-DTPA showed marked en- mandible as has been documented (45).Mirra et al. hancement of the margin and slight enhancement of (10)proposed that these lesions are synovial cysts the inner part of the cyst cavity.This finding was not arising from a developmental juxtaepiphyseal error observed in the contrast-enhanced MRIs of the true with the intraosseous incorporation of synovial tis- cysts with an epithelial lining,which show no enhan- sue. A small nest of synovium becomes trapped cement in the cavity (17, 26).Therefore, contrast- intraosseously during fetal or early infant develop- enhanced MRI can provide useful information for ment and that this tissue may retain some secretory distinguishing TBCs from other cysts and tumors. function, resulting in the development of a cyst. This finding was observed in the present case and a Furthermore,they hypothesized that the fibrous tis- clinical diagnosis of TBC was made. sue and osteoid and giant cells often found at the

Several hypotheses for the pathogenesis of TBC periphery of TBC are from a host bone reaction(10). have been proposed. The myriad of different This theory may explain the greater occurrence in proposed mechanisms provides some insight into the adolescents when developmental anomalies often lack of understanding of this unusual entity. The first present.Similarly,TBCs of the long bones are most frequently proposed theory for the development often discovered at young ages,although this is most of these lesions involves a traumatic event inciting often a result of pathologic fracture.However,the medullary hemorrhage and a subsequent failure of etiology of TBCs remains unclear. the hematoma to organize and be replaced with Histological presentation is classically a vacant tissue(49).Many authors have questioned this mech- cavity of cancellous bone usually unlined or occa- anism,given that often there is no history of trauma sionally lined with a thin connective tissue layer with and,furthermore,the incidence of trauma in patients a scant liquid content.The distinctive characteristic with TBCs is no greater than in the general popula- of TBCs is the absence of epithelial lining.Previous Int J Oral-Med Sci 10(1):31-36,2011 reports have shown that in only 9.52% of the cases cases and proposal for a minimal surgical interven- could a histological evaluation be made of the mate- tion. Int J Pediatr Otorhinolaryngol, 74: 1449-1451, 2010. rial obtained, revealing the presence of vascular 5. Surej Kumar LK, Kurien N, Thaha KA : Traumatic connective tissue without evidence of an epithelial bone cyst of mandible.J Maxillofac Oral Surg, DOI component(49,52).This suggests that the absence of 10.1007/s12663-010-0114-8,2010. 6. Lucas CD,Blum T : Do all cysts in the jaws originate epithelial tissue is one of the most characteristic from the dental system?JADA,16: 647-661,1929. features of TBCs. This finding was found in the 7. Rushton MA : Solitary bone cysts in the mandible.Br present case.In addition,hemosiderin granules were Dent J,81: 37-49, 1946. observed in the cyst wall,suggesting that the episode 8. Schajowicz F, Sissons HA, Sobin LH : The World of hemorrhage was associated with trauma,although Health Organization’s histologic classification of bone she did not recall her antecedent of trauma to the tumors.A commentary on the second edition.Cancer, 75: 1208-1214,1995. anterior mandible. 9. Barnes L, Eveson JW, Reichart P, Sidransky D, edi- Although surgical exploration not only confirms tors. World Health Organization Classification of the diagnosis but also is curative as the curettage Tumours.Pathology and Genetics of Head and Neck performed during the procedure induces bleeding and Tumours.Lyon: IARC Press,2005. 10.Mirra JM, Bernard GW, Bullough PG, Johnston W, further osseous regeneration (50),a high recurrence Mink G : -like bone production in solitary rate of 65.4% (17/26) of cases with a scalloped - bone cysts (so called “”of long bones). margin,smooth margin (4.8%)was reported.There- Report of three cases.Electron microscopic observa- tions supporting a synovial origin to the simple bone fore,a scalloped margin is a sign of possible recur- cyst.Clin Orthop Relat Res,135: 295-307,1978. rence,although this should not be confused with the 11.Hosseini M : Two atypical solitary bone cysts.Br J interdental scalloping associated with an intact - Oral Surg, 16: 262 269, 1978. lamina dura (25).Although careful curettage of the 12.Gilman RH,Dingman RO: A solitary bone cyst of the lesion itself favored bone formation and healing in mandibular condyle.Plastic Reconstr Surg, 70: 610- 614,1982. the present case, the treated lesion should be foll- 13.Pogrel MA : A solitary bone cyst possibly caused by owed up until complete healing has been confirmed removal of an impacted third molar.J Oral Maxillofac radiographically. Surg, 45: 721-723,1987. - 14.Hara H, Ohishi M, Higuchi Y: Fibrous dysplasia of In summary,TBC is an uncommon nonepithelial lined cavity of the jaws.In the clinical diagnosis of the mandible associated with large solitary bone cyst. J Oral Maxillofac Surg, 48: 88-91,1990. TBC,it may be difficult to differentiate TBC from 15.Kuttenberger J,Farmand M,Stoss H : Recurrence of cysts or tumors; however,we obtained the clinical a solitary bone cyst of the mandibular condyle in a diagnosis of TBC using contrast-enhanced MRI. bone graft.Oral Surg Oral Med Oral Pathol,74: 550- 556,1992. 16.Copete MA, Kawamata A, Langlais RP : Solitary

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